Case Report

A 79-year-old caucasian female was admitted due to painless jaundice, choluria, acholia and generalized pruritus for the previous week. On physical examination, an epigastric mass was documented. Abdominal ultrasonography showed dilation of common bile duct (CBD) and intrahepatic biliary tree, with an apparent stricture on CBD middle third. Laboratory findings included microcytic anaemia (Hb 6.5 g/dL), normal inflammatory markers and high levels of serum bilirubin (6.0 mg/dL), elevated transaminases (AST 262 IU/L; ALT 492 IU/L) and cholestasis parameters (GGT 426 IU/L; AF 519 IU/L). An endoscopic ultrasonography (EUS) was performed and showed, on the gastric cardia, an irregular and ulcerated lesion with 4-5cm, extending to the lesser curvature (Figure 1) and involving all gastric wall layers, splenic vessels and pancreas (T4) (Figure 2). Malignant perigastric adenopathies were identified (N+). Pancreas had no other parenchymal or ductal abnormalities. Ampullary area and distal common bile duct were normal. However, on the middle third of the common bile duct, a hypoecogenic, heterogeneous, irregular, infiltrative and stenotic lesion was identified (Figure 3). This lesion caused marked dilation of the proximal common bile duct and intra-hepatic biliary ducts and presented a hard pattern on elastography (Figure 4).

Figure 1 – Irregular and ulcerated lesion with 4-5cm on gastric cardia, extending to the lesser curvature.

Figure 2 – Gastric lesion involving all gastric wall layers, splenic vessels and pancreas (T4).

Figure 3 – Hypoecogenic, heterogeneous, irregular, infiltrative and stenotic lesion on the middle third of the common bile duct.

Figure 4 – Hard (blue) elastographic pattern of the common bile duct lesion.

 

The parenchyma of the left liver lobe was unremarkable. Biopsies confirmed gastric adenocarcinoma (intestinal type, Lauren classification). At this point, the hypotheses of a synchronous cholangiocarcinoma versus a biliary metastasis of the gastric cancer were placed as the cause of the bile duct stricture. An ERCP with direct cholangioscopy (SpyGlass®, Boston Scientific) was then performed. Cholangiography confirmed an irregular stricture of the middle third of the common bile duct with upstream dilation of the biliary tree (Figure 5). Sphincterotomy was done. Cholangioscopy showed a stenotic segment just above the cystic duct insertion, with irregular and friable mucosa with aberrant vessels and biopsies were undertaken (Video). A self-expandable uncovered metal stent 8x60mm was placed (WallFlex®, Boston Scientific) (Figure 6).

Figure 5 – Cholangiography showing an irregular stricture of the middle third of the common bile duct with upstream dilation of the biliary tree.

Video – Cholangioscopy.

Figure 6 – Placement of a self-expandable uncovered metal stent 8x60mm (WallFlex®, Boston Scientific).

 

Biopsies revealed adenocarcinoma and the immunohistochemistry study performed on both gastric and biliary stricture biopsies showed positivity for cytokeratin 19 and 20 and for CDX2 (Figures 7A and 7B). CK7 was negative and the proliferative index (Ki67) was high. Thus, a common bile duct metastasis of a gastric adenocarcinoma was diagnosed (M1).

Figure 7A – Imunohistochemistry of gastric biopsies.

Figure 7B – Imunohistochemistry of common bile duct biopsies.

 

Commentary

Painless jaundice due to intraductal common bile duct obstruction is far more frequently caused by primary biliary or pancreatic cancer than by metastatic disease.1 Extrinsic obstruction due to malignant lymphadenopathies is also much more common.1 Intraductal common bile duct secondary lesions are extremely rare and have been scarcely characterized in the literature. In the few cases described, it occurred in breast, colon and gastric cancer.2,3 Differential CT features suggestive of intraductal metastasis rather than cholangiocarcinoma were described.4 However, EUS and direct cholangiography are probably more useful exams for this differential diagnosis, as they allow a better characterization of the common bile duct strictures and allow tissue acquisition for cytologic/histopathologic examination. In the present case, EUS was essential not only for the diagnosis and staging of the primary tumor, but also for the identification of this rare type of metastasis. EUS has a well-established role in the study of biliary strictures. However, fine-needle aspiration (FNA) has a lower sensivity in this context, mainly if a mass is not detected at EUS.5 Thus, endoluminal sampling is recommended either as an alternative or in combination to FNA.5 ERCP conventional sampling (intrabiliary forceps and cytology brush) is inferior or at maximum, comparable to FNA.6 Cholangioscopy assumes an important role in those cases in which false negatives were obtained with these two techniques.7 Although the risk of FNA seems exceedingly low, cholagioscopy overtakes the concerns about tumor seeding.5

 

References:

1. Lee DH, Ahn YJ, Shin R, Lee HW. Metastatic mucinous adenocarcinoma of the distal common bile duct, from transverse colon cancer presenting as obstructive jaundice. Korean J Hepatobiliary Pancreat Surg 2015;19:125-128.

2. Moon SG, Han JK, Kim TK, Kim AY, Kim TJ, Choi BI. Biliary obstruction in metastatic disease: thin section helical CT findings. Abdom Imaging 2003;28:45–52.

3. Coletta M, Montalti R, Pistelli M, Vincenzi P, Mocchegiani F, Vivarelli M. Metastatic breast cancer mimicking a hilar cholangiocarcinoma: case report and review of the literature, World Journal of Surgical Oncology 2014;12:384.

4. Lee YJ, Kim SH, Lee JY, Kim MA, Lee JM, Han JK, et al. Differential CT features of intraductal biliary metastasis and double primary intraductal polypoid cholangiocarcinoma in patients with a history of extrabiliary malignancy. AJR Am J Roentgenol 2009;193:1061-1069.

5. Dumonceau JM, Deprez P, Jenssen C, Iglesias-Garcia J, Larghi A, van Biervliet G, et al. Indications, results, and clinical impact of endoscopic ultrasound (EUS)-guided sampling in gastroenterology: European Society of Gastrointestinal Endoscopy (ESGE) Clinical Guideline – Updated October 2016. Endoscopy 2016 (in press).

6. Weilert F, Bhat YM, Binmoeller KF, Kane S, Jaffee IM, Shaw RE, et al. EUS-FNA is superior to ERCP-based tissue sampling in suspected malignant biliary obstruction: results of a prospective, single-blind, comparative study. Gastrointest Endosc 2014;80:97-104.

7. Siddiqui AA, Mehendiratta V, Jackson W, Loren DE, Kowalski TE, Eloubeidi MA. Identification of cholangiocarcinoma by using the Spyglass Spyscope system for peroral cholangioscopy and biopsy collection. Clin Gastroenterol Hepatol 2012;10:466-471.

Authors:

Joana Carmo1,2, Carlos Sanchez-Vilar1,3, Francisco Martín-Presas1, José Lariño-Noia1, Júlio Iglesias-García1, Ihab Abdulkader4, Enrique Domínguez-Muñoz1

1. Gastroenterology Department, Hospital Clinico Universitario de Santiago de Compostela, Santiago de Compostela, Spain

2. Gastroenterology Department, Hospital Egas Moniz, Lisboa, Portugal

3. Gastroenterology Department, Complejo Hospitalario Universitario Insular Materno Infantil de Gran Canaria, Las Palmas, Spain

4. Pathology Department, Hospital Clinico Universitario de Santiago de Compostela, Santiago de Compostela, Spain